[A Case Report of Adenomyoepithelioma Coexisting with Apocrine Carcinoma in Contralateral Breasts].

Adenomyoepithelioma(AME)of the breast is a rare condition, and comorbidity with carcinoma is even more unusual. Herein, we report a case of both AME and apocrine carcinoma in different breasts of a single patient. A 48-year-old woman presented to our clinic with a right breast tumor. Fine needle aspiration cytology(FNAC)was indeterminate and suspicious for both papilloma and non-invasive ductal carcinoma, but excisional biopsy indicated an AME. Immuno-histochemical staining showed EMA(+), AE1/3(+), and CK7(+)mammary duct cells and αSMA(+), CK5/6(+), and p63(+) myoepithelial cells. Six months later, the patient noticed a left breast tumor, and although FNAC indicated no malignancy, after 6 additional months, the tumor size had increased and a mammography revealed tumor microcalcification, suggesting malignancy. Vacuum-assisted biopsy revealed an apocrine carcinoma. The patient underwent partial mastectomy and sentinel node biopsy, followed by radiotherapy and chemotherapy. The post-surgical pathology was pT1pN0M0, Stage Ⅰ, triple- negative, and the patient was disease-free for 12 years postoperatively. To our knowledge, this is only the second case of AME and breast cancer in different breasts reported in Japan.

Clinicopathological characteristics and outcomes of malignant adenomyoepithelioma of the breast: a single institution's experience.

BACKGROUND: Malignant adenomyoepithelioma of the breast is a rare tumor and most of relevant literature consists of individual case reports. This study objective was designed to evaluate clinicopathological features and treatment outcomes of 15 cases of malignant adenomyoepithelioma at a single institute. METHODS: A retrospective medical record review was performed for 15 subjects confirmed with malignant adenomyoepithelioma upon postoperative pathological diagnosis at the Asan Medical Center from January 2008 to June 2018. Data regarding age at diagnosis, preoperative biopsy results, operation methods, the status of hormone receptors and HER2, and clinical outcomes were collected. RESULTS: All cases were female patients diagnosed at median age of 50 years. Preoperative core needle biopsy results showed that 40% of the cases (6 out of 15) were benign which was in discordance with the final malignant pathology report. Thirteen cases underwent wide excision with or without sentinel lymph node biopsy (SLNB) and 2 cases had total mastectomy with SLNB. Five of 11 cases (45.5%) were triple negative. Ten of 15 cases underwent postoperative radiation therapy, 3 cases underwent chemotherapy, and 5 cases underwent endocrine therapy. During median follow-up of 55 months, the 5-year overall survival rate was 87.5% and the 5-year disease free survival rate was 91.7%. Two lung metastases developed. One case showed local recurrence 3 years after surgery and radiotherapy and subsequently developed lung metastasis 1 year late. Another case developed lung metastasis one and a half years after surgery in combination with endocrine therapy and neoadjuvant chemotherapy. CONCLUSION: Preoperative core needle biopsy showed inaccurate results for diagnosing malignant adenomyoepithelioma. Malignant adenomyoepithelioma has a high rate of triple negative subtype but has a relatively good prognosis although there is a risk of local and systemic recurrence.

Adenomyoepithelioma of the breast with prominent cystic changes: a case report.

BACKGROUND: Adenomyoepithelioma (AME) of the breast is a rare subtype of breast tumor. Most of AMEs reported are solid, however, cystic or prominent cystic changes are extremely rare. CASE PRESENTATION: A 51-year-old woman presented a lump in the upper outer quadrant of right breast, and it was accompanied by continuous breast pain and bilateral axillary itching for more than 2 months. There were no other symptoms found. Preoperative mammography and ultrasound examination were performed. Mammography showed a noncalcified lobulated mass, and it was considered to be a benign cyst with septum on ultrasound, but ductal carcinoma of breast, adenoid cystic carcinoma could not be excluded. At first, AME was not considered preoperatively, because the imaging features of this rare tumor may vary widely, which may result in an incorrect diagnosis. But eventually, AME was diagnosed by postoperative pathology and immunohistochemistry. CONCLUSION: We herein present a rare case of breast AME with prominent cystic changes. AME has no-specific imaging features, but the benign or malignant nature of the lesion might be suspected on imaging.

Clinical presentation, national practice patterns, and outcomes of breast adenomyoepithelioma.

Breast adenomyoepithelioma (AME) is a rare tumor with the published literature mainly in the form of case reports. Thus, there is currently only limited published data to guide evidence-based management. We sought to use a large, contemporary US database to evaluate how these patients are managed and describe expected outcomes. The National Cancer Database was queried (2004-2013) for women with AME. Statistics included multivariable logistic regression, Kaplan-Meier analysis to evaluate overall survival (OS) and Cox proportional hazards modeling. Overall, 110 patients were analyzed. At diagnosis, the median age was 67 years and the median tumor size was 2.0 cm. All but four patients had node-negative disease. A majority (55%) of tumors were estrogen receptor negative, and only one was positive for HER2/neu. The most common surgical procedure was lumpectomy (60%); a minority (10.9%) of subjects underwent complete axillary nodal dissection, with one-quarter not undergoing pathologic nodal sampling. Chemotherapy, hormonal therapy, and radiotherapy were utilized in a minority of patients (26%, 8%, and 36%, respectively), and none were associated with OS. At median follow-up of 52 months, the 5-year OS for the entire population was 74.4%. Disease-related characteristics and practice patterns are described for AME, the largest study of this rare tumor to date. Resection is the most important aspect of management, and based on this dataset the low rate of nodal involvement suggests that in some cases nodal sampling could be safely omitted. Adjuvant therapy may be considered on a case-by-case basis. Taken together, these data provide valuable insight into a rare neoplasm that may better inform management of these patients.

Adenomyoepithelioma of the breast: case report and literature review.

Adenomyoepithelioma are uncommon tumors. The majority of them occur in women in the fifth and sixth decades who usually present with a self-palpated, solitary breast mass or a lesion identified on mammography. We report the case of adenomyoepithelioma of the breast with malignant transformation of both myoepitheliel and epithelial components diagnosed as malignancy during the preoperative stage in a seventy-six year old woman.

A Case of Adenomyoepithelioma of the Breast Showing Strong Uptake of 18 F-Fluorodeoxyglucose on a Positron Emission Tomography.

An adenomyoepithelioma of the breast is a rare tumor characterized by biphasic proliferation of both epithelial and myoepithelial cells. This tumor is generally considered as a benign neoplasm, and there are few reports describing the imaging features of this tumor through 18 F-fluorodeoxyglucose positron emission tomography (FDG-PET). Here, we report a case of an adenomyoepithelioma that showed strong uptake of FDG on PET similar to that observed with a malignant tumor. A 73-year-old woman presented to our hospital with a 3.5-cm, mobile, and elastic hard tumor in the upper area of the left breast. Although the findings of mammography, ultrasonography, and contrast-enhanced magnetic resonance imaging suggested that the tumor was malignant, it was diagnosed as an adenomyoepithelioma by core needle biopsy. An invasive ductal carcinoma, 0.5-cm in size, was detected in the medial upper area of the ipsilateral breast during an examination. Although FDG-PET demonstrated no lymph node or distant metastases from the invasive ductal carcinoma, strong uptake of FDG was detected in the adenomyoepithelioma. Breast conserving surgery and sentinel lymph node biopsy for the invasive ductal carcinoma together with resection of the adenomyoepithelioma was performed. A diagnosis of adenomyoepithelioma was confirmed through histologic examination of the resected specimen. This case indicates that some adenomyoepitheliomas may show a strong uptake of FDG on PET, which resembles a malignant tumor.

Cutaneous Adenomyoepithelioma: Report of a Case and Review of the Literature.

Adenomyoepithelioma (AME) is a biphasic neoplasm of epithelial and myoepithelial cells. It is most commonly found in the breast, although rare cases have been reported from the lung, salivary glands, and skin. There are 5 well-documented cases of cutaneous AME in the literature. We report a new case of cutaneous AME. Our case was commingled with apocrine hidrocystoma. This is the first report of cutaneous AME in a male patient and the first to describe SOX10 immunostaining in cutaneous AME. We review the literature on cutaneous AME and note the greater than chance colocalization with other adnexal tumors. We speculate that AME may represent localized overgrowth of myoepithelial cells within a pre-existent sweat gland tumor. Histopathologists should be aware of the potential of SOX10-positive myoepithelial neoplasms to mimic nodular melanocytic proliferations.

Epithelial-myoepithelial carcinoma of the minor salivary glands: a case report and review of the literature.

Epithelial-myoepithelial carcinoma (EMC) is a rare tumor, especially in the minor salivary glands. This case report describes a 40-year-old man who presented with an EMC on the palate. Histologically, the tumor was characterized by a biphasic structure consisting of duct-lining cuboidal cells in the inner layer and clear myoepithelial cells in the outer layer. The clear cells predominated, and the diagnosis was confirmed with immunohistochemical staining. The inner cuboidal epithelial cells were positive for cytokeratins, whereas the outer cells were positive for S-100 protein and smooth muscle actin. The patient was treated with a wide surgical excision, and no recurrence or metastasis was observed at a 10-year follow-up. A literature review found only 30 well-documented cases of EMC in the intraoral minor salivary glands.

[Adenomyoepithelioma with dominant myoepithelial contingent of the breast: A case report and literature review]. / Adénomyoépithéliome avec contingent myoépithélial prédominant du sein : à propos d'une observation et revue de la littérature.

We report a case of adenomyoepithelioma with predominant myoepithelial quota, a rare tumor of the breast due to proliferation of epithelial and myoepithelial cells in a patient of 71 years. This lesion, with difficult radiological and pathological diagnosis (biopsy) in the initial stage of the treatment, should benefit from surgical resection in healthy margin. In fact, this tumor is evolving in most cases on a benin mode, but cases of local or metastatic recurrences were reported. Histological and immunohistochemical arguments are important to reach the final diagnosis.

Benign Adenomyoepithelioma: An Unrecognised Precursor of Ductal Carcinoma in Situ in Patient With Lynch Syndrome.

Currently, there is no robust evidence demonstrating a clear association between Lynch syndrome and non-malignant breast pathology such as adenomyoepithelioma. We report a case of benign breast adenomyoepithelioma, which after recurrence was associated with ductal carcinoma in-situ (DCIS) in a 41-year-old woman with Lynch syndrome, who lacked significant family history of breast or ovarian cancer. Both, the adenomyoepithelioma and DCIS were found to have nuclear loss of MSH2/MSH6 by immunohistochemistry, while germline testing confirmed MSH2 gene mutation. Concordant loss of MSH2 in both lesions in the context of a MSH2 pathogenic variant in this patient with Lynch syndrome illustrates that the benign adenomyoepithelioma behaved as a likely precursor of DCIS. Our report provides a novel perspective that in some patients with Lynch syndrome adenomyoepithelioma may represent a pre-malignant precursor lesion of DCIS.

Adenomyoepithelioma of the Breast: A Report of 3 Cases.

BACKGROUND Breast adenomyoepithelioma is a rare benign breast tumor characterized by a biphasic proliferation of epithelial and myoepithelial cells with variable clinical and diagnostic features. Establishing the diagnosis, determining optimal therapy, and predicting outcome are problematic because of the rarity of this entity. There have been only 2 large series of adenomyoepitheliomas of the breast, reported by Tavassoli and Rosen, which included 27 and 18 patients, respectively. In this report, we present 3 cases of breast adenomyoepithelioma. CASE REPORT Herein, we report 3 cases of breast adenomyoepithelioma. The first case is of a 64-year-old woman who was found to have right breast microcalcification on a screening mammogram. The second case is of a 74-year-old woman who had a right breast mass. These 2 patients were managed by wide local excision. Postoperative microscopic examination revealed adenomyoepithelioma. The third case is of a 49-year-old woman with bilateral saline breast implants who presented with a left breast mass. A core needle biopsy was done and revealed adenomyoepithelioma associated with usual ductal hyperplasia and ductal carcinoma in situ. CONCLUSIONS Breast adenomyoepithelioma is a rare condition that can pose diagnostic challenges due to variable imaging presentations, necessitating percutaneous core biopsy for initial diagnosis. Correct diagnosis is usually possible only on excisional biopsy and confirmed by demonstrating the biphasic nature of the tumor by IHC. Clinical suspicion coupled with utilizing both radiological and histopathological facilities can aid in the accurate diagnosis and management. For the most part, they are considered to be benign, but they can locally recur.

Benign adenomyoepithelioma of the breast: a case report.

The diagnosis of adenomyoepitheliomas is difficult and relies on the presence of a double component of epithelial and myoepithelial cells belonging to the breast lobules and ducts. The clinical and imaging characteristics are not specific; thus, the diagnosis is histological. In this article, we present a case of a young female who presented with a 2 cm lump in the breast without other clinical symptoms, which revealed a benign adenomyoepithelioma (AME). We performed a large excisional lumpectomy, and the patient recovered well with no complication or recurrence within two years follow-up. When it comes to adenomyoepitheliomas, the published literature is mainly composed of case reports, so much so that there are no evidence-based guidelines. Our case shows that an excisional lumpectomy is often enough when facing a small size tumor with no signs of malignancy, which contributes to the limited data on the subject.

Adenomyoepithelioma of the breast: a rare diagnosis complicated by surgical emergency and diagnostic uncertainty.

A woman in her 80s was referred as an emergency case with a large oedematous and ulcerating lesion of the right breast. There was a 5-month history of increasing breast volume with new onset skin breakdown and discharge. Imaging revealed an extensive heterogeneous mass requiring drainage. No diagnosis was received from multiple biopsies and immediate surgical resection of the breast and axillary sampling was prioritised given the deteriorating patient condition. Postoperative histology identified a biphasic Adenomyoepithelioma of low malignant potential, a rare presentation compounding the complexity of management. The diagnostic uncertainty of this case highlighted the importance of MDT collaboration and the flexibility of current management pathways when dealing with cases requiring urgent surgical intervention. Axillary sampling in the context of unsuccessful preoperative biopsy represented a comprehensive means for assessing the need for further surgical or systemic management in the context of unconfirmed malignancy in a deteriorating patient.

Malignant Adenomyoepithelioma of the Breast and Associated Epithelial-Myoepithelial Carcinoma; A Rare Case Report.

Adenomyoepithelioma comprises a spectrum of lesions with variable morphology and clinical behavior, presenting at a wide age range. The most common presenting symptom is palpable abnormality. Mammographic abnormalities include focal asymmetries, masses and microcalcifications. Adenomyoepithelioma is a biphasic neoplasm characterized by proliferation of epithelial and myoepithelial cells. Adenomyoepitheliomas can be benign, atypical and malignant (adenomyoepithelioma with carcinoma). Malignant transformation occurs in either one or both cellular components leading to the development of invasive carcinoma. Invasive carcinoma types include invasive breast carcinoma of no special type, invasive lobular carcinoma, invasive carcinoma of special types, myoepithelial carcinoma, metaplastic carcinoma and biphasic carcinoma such as epithelial-myoepithelial carcinoma. While the majority of classic adenomyoepitheliomas have a benign clinical course and can be treated by local excision, local recurrence and distant metastasis have been reported. In malignant cases, treatment is determined by the associated carcinoma to include radiotherapy after breast conserving surgery and sentinel lymph node biopsy or axillary lymph node dissection, as indicated. Herein we report a case of a 62 year old woman who was found to have focal asymmetry on screening mammogram. She underwent a core biopsy of the lesion which showed atypical epithelial-myoepithelial neoplasm and excision was recommended. Upon excision, a diagnosis of malignant adenomyoepithelioma with associated epithelial-myoepithelial carcinoma was rendered with negative margins. The patient declined additional surgery for sentinel lymph node biopsy and declined adjuvant therapy. Six months after surgery, the patient is doing well with no complains. A follow-up mammogram and ultrasound of the axilla showed no abnormalities.

Primary mucoepidermoid carcinoma of the breast arising in adenomyoepithelioma.

Mucoepidermoid carcinoma (MEC) and adenomyoepithelioma (AME) are uncommon neoplasms of the breast that are more commonly noted in the salivary glands. AMEs are benign tumours that are known to undergo malignant transformation. This report describes the first case of a MEC arising in AME in a woman in her 50s.

Adenomyoepithelioma of the breast with unusual confounding diagnostic feature: a case report.

BACKGROUND: Adenomyoepithelioma of the breast is an uncommon subtype of breast neoplasm that occurs in adults over a wide age range but most commonly in middle-aged and older adults. It usually presents as a solitary palpable mass or is detected on breast radiographic images. Histologically, it is a biphasic tumor with proliferation of both the epithelial and myoepithelial components of the glands, with variable types of tissue metaplasia. CASE PRESENTATION: A 64-year-old Saudi woman who underwent regular breast screening (mammogram) presented to our hospital following radiographic detection of a suspicious grouped microcalcification in the upper outer quadrant of her right breast on the mammogram. A wide local excision of the right breast lump was performed. Following histopathological examination of the breast lump, the final diagnosis was breast adenomyoepithelioma with mucoepidermoid/divergent differentiation, with no evidence of malignancy. About two years after the operation, a clinical follow-up conducted outside our hospital showed the development of ductal carcinoma in situ in the same breast. CONCLUSION: Although the prognosis and the plan of treatment remains the same, our case highlights the complexities in making an accurate diagnosis between the various types of metaplasia within adenomyoepithelioma on one hand and the presence of mucoepidermoid differentiation in adenomyoepithelioma on the other.

[A case of breast adenomyoepithelioma that was diagnosed by local excision].

A case is a 72-year-old woman with right breast lump detected by palpation. In the end of November 2009, we palpated a mass with clear boundaries in the right D area. Breast echography showed a nearly 3 cm tumor in the cyst. MRI showed an augmented effect of marginal irregular 3 cm in size. A diagnosis was adenomyoepithelioma by needle biopsy. We performed lumpectomy because a breast cancer was suspected. Histopathology findings were adenomyoepithelioma ER(+), PgR(-), HER2(0), α SMA(+). A diagnosis of adenomyoepithelioma is rather hard by means of imaging or biopsy. When this disorder was in doubt, a tissue analysis is indispensable for a determination of diagnosis because we were able to make a diagnosis by local excision without an excessive invasion. So we herein report our experience of adenomyoepithelioma with reference.